Unit of Clinic of Central and Peripheral Degenerative Neuropathies, IRCCS Foundation, C. Besta Neurological Institute, Milan, Italy.
Neuromuscul Disord. 2011 Feb;21(2):129-31. doi: 10.1016/j.nmd.2010.09.009. Epub 2010 Oct 14.
Mitofusin-2 gene (MFN2) mutations cause Charcot-Marie-Tooth type 2A (CMT2A), sometimes complicated by additional features such as optic atrophy, hearing loss, upper motor neuron signs and cerebral white-matter abnormalities. Here we report, for the first time, the occurrence of motor neuron disease, consistent with amyotrophic lateral sclerosis (ALS), in a 62-year-old woman affected by early-onset slowly progressive CMT2A, due to a novel MFN2 mutation. After age 60, rate of disease progression changed and she rapidly developed generalised muscle wasting, weakness, and fasciculations, together with dysarthria and dysphagia. Clinical features, EMG findings, and fast progression were consistent with ALS superimposed on CMT.
线粒体融合蛋白 2 基因 (MFN2) 突变导致 2A 型腓骨肌萎缩症 (CMT2A),有时伴有其他特征,如视神经萎缩、听力损失、上运动神经元征和脑白质异常。这里我们首次报告了一例 62 岁女性患有早发性进行性 CMT2A,由于一种新的 MFN2 突变,导致运动神经元病,符合肌萎缩侧索硬化症 (ALS)。60 岁后,疾病进展速度改变,她迅速出现全身性肌肉萎缩、无力和肌束震颤,以及构音障碍和吞咽困难。临床特征、肌电图结果和快速进展与 ALS 合并 CMT 一致。